KBB ‹htisas Dergisi
2002;9(2):132-137
OLGU SUNUMU
Laryngotracheal reconstruction of the congenital
glotto-subglottic stenosis with autogenous
thyroid cartilage interposition: a case report*
Do¤ufltan glotto-subglottik stenozun otojen tiroit kartilaj interpozisyonu ile
larengotrakeal rekonstrüksiyonu: Bir olgu sunumu
Orhan ÖZTURAN,1 Ahmet KIZILAY,1 Murat Cem M‹MAN,1
Semih ÖNCEL,2 M. Tayyar KALCIO⁄LU,1 Tamer ERDEM1
Surgical correction of grade III glotto-subglottic stenosis
in a two-month-old girl was illustrated in a staged manner. Firstly, a silicone keel was placed via anterior thyrotomy following a tracheotomy. Secondly, laryngotracheal reconstruction was performed by interposing an
autogenous thyroid cartilage anteriorly between the
edges of the longitudinally divided cricoid cartilage and
the upper tracheal rings. A stent was maintained for two
months. The glottis and subglottis appeared patent and
healed following removal of the stent. A meaningful
voice and rather comfortable respiration were observed
during a 13-month follow-up.The use of thyroid cartilage
autograft offers many advantages in laryngotracheal
reconstruction with considerably less technical difficulty.
Grade III do¤ufltan glotto-subglottik stenozlu iki ayl›k
bir k›z bebekte aflamal› cerrahi tedavi uygulad›k. ‹lk
aflamada, trakeotomiyi takiben anterior tirotomi yoluyla silikon bir kil yerlefltirildi. ‹kinci aflamada, önde
ve orta hatta longitudinal olarak kesilen krikoid ve
üst trakeal halkalar›n kenarlar› aras›na otojen tiroit
kartilaj greftinin interpozisyonu ile larengotrakeal rekonstrüksiyon yap›ld›. ‹ki ay süreyle stent uyguland›.
Stentin al›nmas›n› takiben glottis ve subglottisin aç›k
ve iyileflmifl oldu¤u görüldü. On üç ayl›k izlem s›ras›nda anlaml› bir ses ve oldukça rahat solunum gözlendi. Larengotrakeal rekonstrüksiyonda tiroit kartilaj
otogreftinin kullan›m›, önemli derecede teknik kolayl›klar yan› s›ra birçok avantaj sa¤lamaktad›r.
Key Words: Cartilage/transplantation; laryngeal cartilages/transplantation; laryngeal diseases/surgery/congenital; laryngostenosis/surgery/congenital; tracheal stenosis/surgery; transplantation, autologous.
Anahtar Sözcükler: Kartilaj/transplantasyon; larenjeal k›k›rdaklar/transplantasyon; larenjeal hastal›klar/do¤ufltan/cerrahi; larengostenoz/do¤ufltan/cerrahi; trakeal stenoz/cerrahi; transplantasyon, otolog.
Laryngeal webs mostly occur at the glottic level
and re p resent a recanalization failure of the larynx
during prenatal development, frequently having a
subglottic extension.[1] Congenital subglottic stenosis
is the third most common congenital laryngeal anomaly following laryngomalacia and vocal cord paraly-
◆
Departments of Otolaryngology, 1 Medicine Faculty of ‹nönü University,
Malatya; 2Atatürk Training and Research Hospital, ‹zmir, both in
Turkey.
◆
1
◆
Received: November 14, 2001. Accepted for publication: January 15, 2002.
◆
Dergiye gelifl tarihi: 14 Kas›m 2001. Yay›n için kabul tarihi: 15 Ocak 2002.
◆
Correspondence: Dr. Orhan Özturan. ‹nönü Üniversitesi, T›p Fakültesi,
KBB Anabilim Dal›, 44300 Malatya, Turkey.
Tel: +90 422 - 341 06 60 / 4602 Fax: +90 422 - 341 07 28
e-mail: [email protected]
◆
‹letiflim adresi: Dr. Orhan Özturan. ‹nönü Üniversitesi T›p Fakültesi
KBB Anabilim Dal›, 44300 Malatya.
Tel: 0422 - 341 06 60 / 4602 Faks: 0422 - 341 07 28
e-posta: [email protected]
* Presented at the 26th National Congress of Turkish Otorhinolaryngology,
Head and Neck Surgery (September 22-26, 2001, Antalya, Turkey).
‹nönü Üniversitesi T›p Fakültesi Kulak Burun Bo¤az Hastal›klar›
Anabilim Dal›, Malatya; 2Atatürk E¤itim ve Araflt›rma Hastanesi Kulak
Burun Bo¤az Klini¤i, ‹zmir.
* 26. Ulusal Türk Otorinolarengoloji ve Bafl-Boyun Cerrahisi Kongresi’nde
sunulmufltur (22-26 Eylül 2001, Antalya).
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Özturan et al., Laryngotracheal re c o n s t ruction of the congenital glotto-subglottic stenosis with autogenous thyroid cartilage interposition
sis.[2] The diagnosis is suspected in a child who presents with an abnormally shaped cricoid, without a
[3]
previous history of intubation.
goscopic blade under magnified vision of an operating
microscope. During direct laryngeal examination,
microscopic and rigid endoscopic views and instrumental manipulation disclosed extension of the glottic
web causing subglottic stenosis. Because the glottic
web prevented the use of an endotracheal tube
through the glottic aperture, a rigid endoscope, 4 mm
in diameter, was inserted through glottic webbing to
determine the size of the subglottic stenosis. Subglottic
airway was assessed as grade III according to the
Myer-Cotton’s classification system.[7]
Insertion of a keel between the divided vocal cords
is a well-accepted surgical treatment of a glottic web.
For subglottic stenosis, laryngotracheal reconstruction
has become the standard treatment in the pediatric
population. A number of surgical techniques have
been developed to deal with this condition. Grafting
of the exposed larynx and trachea with cartilage provides healing and a speedy extubation or decannulation. Costal or auricular conchal cartilages have
become the most common grafting materials used to
expand the airway through open surgical procedures.
However, donor site discomfort and morbidity are
potential problems when either costal or auricular cartilage is used in laryngotracheal procedures.[4]
Due to the inherent risks for granulation tissue
formation at the glottic level and aspiration, a staged
p ro c e d u re was performed. In the first stage, a
Montgomery silicone laryngeal keel (Boston Medical,
Westborough, MA, USA) was placed between the
divided vocal cords through anterior thyrotomy to
provide smooth healing of the glottic web. The intubation tube inserted via tracheotomy was removed
and a tracheotomy tube was introduced. Respiration
was maintained through this tracheotomy tube for six
weeks, with the silicone keel placed between the
vocal cord s .
Recently, the beneficial use of autogenous thyroid
cartilage graft for the expansion of the subglottic
region of the larynx have been demonstrated in animal models.[5,6] This article presents the application of
this technique in an infant with grade III glotto-subglottic stenosis.
In the second stage, the keel was removed externally without reopening the thyroid cartilage at the
time of laryngotracheal reconstruction. Direct laryngoscopy showed the separated and healed, but edematous vocal cords (Fig. 1b) and a 75% occlusion of
the subglottic space, mostly by cartilaginous shelves
anteriorly and laterally (Fig. 2a). A vertical incision
was made in the skin over the laryngotracheal stru ct u res and the strap muscles were divided along the
median raphe. After an adequate exposure, a vertical
midline incision was made over the cricothyroid
membrane, the cricoid cartilage, and the first two tracheal cartilages extending down to the tracheotomy
site. The posterior cricoid plate was also divided in
the midline to the posterior perichondrium of the
cricoid cartilage with the use of a number 15 blade.
Adequate enlargement of the subglottic lumen by
anterior and posterior incisions obviated lateral divisions. A fusiform-shaped cartilage graft, 2x1 cm in
size, was removed from the superior aspect of the
right thyroid lamina (Fig. 3a), with special attention
given to leave the inner layer of the perichondrium
intact. The outer layer of the perichondrium was harvested with the graft (Fig. 3b). Another piece of cartilage, small in size, was inserted between the divided
posterior cricoid plate and was fit flush between the
divided posterior cricoid lamina, which was then
CASE REPORT
A two-month-old girl presented with manifestations of aphonia, biphasic stridor, suprasternal re t r a ctions, and cyanotic episodes which had made nursing
difficult since birth. No history of prescribed medications and intubation were elicited for the mother and
the infant, respectively. She was born to consanguineous parents; however, other siblings did not have
any evidence of an anomaly. The mother was delivered
at 32 weeks of gestation. A complete physical examination was undertaken to exclude any associated
anomalies including atrial or ventricular septal defects.
Flexible laryngoscopy showed a glottic web along the
membranous cord. Tracheotomy was performed at the
level of the third tracheal cartilage with a vertical incision under general anesthesia through a face mask.
Then, anesthesia was maintained through an intubation tube inserted via tracheotomy. Laryngeal stru ctures were examined by direct laryngoscopy, which
revealed a thick web involving 70% of the glottis. The
individual vocal folds seemed to undergo fusion. A
diagnosis of type III congenital glottic web was made
(Fig. 1a).[1] The glottic web was incised endoscopically
with precision prior to an anterior thyrotomy
approach. The incision was made in the midline up to
the anterior commissure with the use of a microlaryn133
Özturan et al., Laryngotracheal re c o n s t ruction of the congenital glotto-subglottic stenosis with autogenous thyroid cartilage interposition
fixed with 6-0 Vicryl sutures placed only to the cut
ends of the posterior cricoid cartilage plate. The knots
w e re buried under the mucosal layer.
glottis appeared patent and healed (Fig. 1b and 2b). The
patient was administered cisapride 0.6 mg/kg/daily
divided into three doses at approximately equal intervals from the time of diagnosis to six months after the
laryngotracheal reconstruction to avoid adverse effects
of gastroesophageal reflux on healing. A meaningful
voice and rather comfortable respiration were observed
during a 13-month follow-up.
A Montgomery T-tube (Boston Medical,
Westborough, MA, USA) with an inner diameter of 6
mm was placed into the lumen. The level of the upper
limp was shortened so that it can lie at a lower level
than that of the glottis, with the external limp extending out from the tracheotomy site. The harvested thyroid cartilage autograft was interposed between the
divided cricoid and upper tracheal cartilages, with
the perichondrial layer facing the airway lumen and
covering the upper limp of the Montgomery T-tube
(Fig. 3b). The graft was fixed in position with the use
of 6-0 polypropylene figure-of-8 sutures. The strap
muscles were reunited with absorbable sutures in the
midline and a contact between the strap muscles and
autogenous thyroid cartilage was established. The
patient’s weight was 5.5 kg at the time of laryngotracheal reconstruction. A circular granulation tissue
formation which appeared in the skin surrounding
the external limp of the T-tube was treated effectively
at two different times by topical application of mitomycin-C (0.4 mg/ml) solution.
DISCUSSION
Congenital subglottic stenosis should be considered in a newborn with no history of airway manipu[8]
lation. Subglottic stenosis occurs when the airway
lumen of the cricoid region measures less than 4 mm
in a full-term infant and 3.5 mm in a preterm infant.[8]
In our case, the airway lumen was 2 mm and was
located posteriorly. Not all patients with subglottic
stenosis can be managed with a standard treatment
plan. Each case must be individually evaluated and
the degree of the stenosis, associated laryngotracheal
pathologies, such as the congenital glottic web in our
case, and the general medical condition of the patient
should be considered prior to giving a decision for
reconstruction.
After two months, the stent was removed and the
patient was intubated via tracheotomy for the final
direct laryngoscopic evaluation. The glottis and sub-
Watchful waiting with tracheotomy alone has
been advocated by some authors, hoping that a spontaneous growth of the stenotic laryngeal lumen may
occur.[9] This approach may be appropriate for mild
(a)
(a)
(b)
(b)
Fig. 1 - (a) Grade III congenital glottic web. Seventy percent
of the glottis was involved by a thick web, extending
into the subglottis. The individual vocal folds were
fused. (b) Direct laryngoscopic view of the separate
and healed, but edematous vocal cords.
Fig. 2 - (a) Grade III congenital subglottic stenosis. Seventy-five
percent of the subglottic space was occluded mostly by
cartilaginous shelves anteriorly and laterally. (b) Direct
laryngoscopic view of the subglottis right after the
removal of the stent.
134
Özturan et al., Laryngotracheal re c o n s t ruction of the congenital glotto-subglottic stenosis with autogenous thyroid cartilage interposition
cases in which growth may be anticipated to create an
airway sufficient to meet the needs of the child.
However, prolonged tracheotomy is not without
problems; it may cause some tracheal damage at the
stoma and tip of the tube, and a tracheocutaneous fistula.[8] Decannulation of the patient is not easy and
some problems may arise. The physical and mental
dependence makes decannulation diff i c u l t .[8] The
existence of a tracheotomy tube can inhibit vocalization and subsequent language development that may
also result in decreased efforts for language stimulation by the parents. In addition to these morbid conditions, mortality rates ranging from 6% to 24% have
been reported for young children with tracheotomy,
whose airway is significantly obstructed above the
[10]
tube as with in our case. Thus, it is advised that
laryngotracheal reconstruction be performed as early
as possible, utilizing surgical techniques that will
enable early decannulation.[11] In our case, we preferred early enlargement surgery to conservative
waiting taking into consideration the severity of the
subglottic stenosis (grade III), glottic web (grade III),
and the inherent risks of the conservative treatment.
cheal reconstruction, this amount seems to be rather
arbitrary.[12] There are no age limitations for laryngotracheal reconstruction, but a minimum weight of 6
[13]
kg is needed before surgery. We had to resort to
s u rgery in our patient at a time her weight was 5.5 kg,
little less than the minimum desirable value.
Montgomery T-tubes may be relatively safe in children, providing that both postoperative and home care
[14]
are meticulously undertaken. It has the advantage of
serving both as a stent and a tracheotomy tube.[15] The
stent was used to minimize the movement of the laryngeal segment induced by normal swallowing, respiration, and daily activities which enhance graft viability
and decrease granulation tissue formation.[12] On the
other hand, it has a potential risk for obstruction, especially when the inner diameter is small, as was in our
case. Since insertion of a posterior graft shortens the
duration for stenting (from 6 months or longer to a 4[16]
to 8-week period) and because of the potential risk
for plugging of the T-tubes having a small diameter,
the stent was kept for only two months after surgery.
The family was made aware of a possible risk for
obstruction and was trained during hospitalization for
four weeks following laryngotracheal reconstruction.
The external limp of the tube was continuously kept
Although a minimum of 10 kg body weight is
desirable for most children undergoing laryngotra-
(a)
(b)
figure-of-8
suture
T-tube
Fig. 3 - (a) A vertical midline incision was made over the cricothyroid membrane, the
cricoid cartilage and the upper tracheal rings. A fusiform-shaped cartilage graft
was removed from the superior aspect of the right thyroid lamina, leaving the
inner layer of perichondrium intact; (b) The thyroid cartilage autograft harvest ed was placed between the divided cricoid and the upper tracheal cartilages using
figure-of-8 sutures, with the perichondrial layer facing the airway lumen and
covering the upper limp of the Montgomery T-tube.
135
Özturan et al., Laryngotracheal re c o n s t ruction of the congenital glotto-subglottic stenosis with autogenous thyroid cartilage interposition
plugged to prevent crust formation within the stent
lumen, with proper suctioning and humidification.
Since the glottic web was treated with a silicone keel
and complete healing was obtained, the upper limp of
the T-tube was placed below the level of the vocal
cords to eliminate the possibility of aspiration and formation of granulation tissue.
including graft failure or migration, pneumonia,
pneumothorax, wound infection or seroma, or subcutaneous emphysema occurred in our case.
Possible disadvantages of the thyroid cartilage
graft include limitations such as that related to the
size of the graft, the likelihood of destabilization of
the laryngeal skeleton, and altered growth of the thyroid lamina. We did not observe donor site morbidity
in our patient up to 13 months after surgery. The graft
was well-secured with figure-of-8 sutures of 6-0
p o l y p ropylene monofilament, keeping the knots
away from the respiratory lumen. This type of suture
prevented the graft and laryngotracheal structures
from moving in a telescopic manner, a point which
has not hitherto been mentioned. There are concerns
regarding the strength of the thyroid cartilage following graft removal and the effect on subsequent laryngeal growth. Park and Forte[5] showed in kittens that
removal of the superior portion of the thyroid alar
cartilage had no apparent effect on subsequent laryngeal growth and stability. Conversely, the inner perichondrium of the thyroid cartilage should be preserved at the donor site for its potential to induce new
cartilage generation.
Grade III subglottic stenosis usually requires an
open intervention to establish an airway sufficient to
permit decannulation.[12] An autologous tissue must
be inserted to maintain the structural integrity. The
ribs and the auricles are the most commonly used
sources for augmentation grafts. Autogenous costal
cartilage grafts present both potential morbidity such
as pneumothorax and technical difficulty in sewing
the graft in the desired position.[17] On the other hand,
harvesting choncal cartilage graft from the auricle
may result in donor site deformity and morbidity. In
a series of 46 procedures in which auricular choncal
grafts were used, occurrence of donor site infection
and postauricular keloids were reported in one and
two cases, respectively.[17]
Pedicled thyroid alar cartilage was proposed as a
graft material for cricoid expansion, first by Fearon and
[18]
Cotton. Although the idea of using a pedicled muscle graft with its own blood supply and mucoperichondrium is attractive, it may not prove to be a good
surgical choice due to small size of the structures.
In conclusion, the use of thyroid cartilage autograft for laryngotracheal reconstruction in our patient
enabled successful decannulation. This technique
proved useful and provided many advantages such
as a single operation site, minimal graft remodelling,
convenient size, shape, and ease in trimming and
suturing the cartilage graft to laryngotracheal stru ct u res, and significantly less technical difficulty in
obtaining the graft. This technique was first described
by Park and Forte[5] and de Jong et al.[6] for laryngotracheal reconstruction in animal models. We demonstrated that it may also be employed in an infant with
a stenotic airway. The lack of the need for additional
incisions and operation site, and fewer potential complications are obvious benefits of the thyroid cartilage
graft in comparison with the use of auricular or costal
cartilage grafts. Yet, the use of the autogenous thyroid
cartilage graft technique should be validated by further studies in cases with acquired stenosis.
Autogenous thyroid cartilage was utilized in animal models for laryngotracheal reconstruction, with
comparable advantages to those of auricular and
costal cartilage grafts, including viability of the thyroid cartilage, need for a single incision and a single
operative site.[5,6] In addition, the thickness of the
superior thyroid cartilage is almost identical to that of
the cricoid cartilage, which decreases the possibility
of graft prolapse into the airway. The thyroid cartilage is firm and it can easily be carved and shaped.[6]
Its outer aspect covered by the perichondrium can be
positioned facing the lumen, providing a convenient
surface for epithelization that can eliminate granulation tissue formation, preventing graft infection, and
improving survival of the cartilage graft.[19] A contact
area with the strap muscles is established, providing
a bridge for nutrients to the graft.[12] During the procedure, extreme care is necessary to ensure a single
needle pass through the graft to avoid necrosis of the
cartilage along the suture tracts.[20] With an appro p r iate technique and precautions, no complications
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